A multi-stem cell basis for craniosynostosis and calvarial mineralization.

ABSTRACT: Craniosynostosis is a group of disorders of premature calvarial suture fusion. The identity of the calvarial stem cells (CSCs) that produce fusion-driving osteoblasts in craniosynostosis remains poorly understood. Here we show that both physiologic calvarial mineralization and pathologic calvarial fusion in craniosynostosis reflect the interaction of two separate stem cell lineages; a previously identified cathepsin K (CTSK) lineage CSC¹ (CTSK⁺ CSC) and a separate discoidin domain-containing receptor 2 (DDR2) lineage stem cell (DDR2⁺ CSC) that we identified in this study. Deletion of Twist1, a gene associated with craniosynostosis in humans^2,3, solely in CTSK⁺ CSCs is sufficient to drive craniosynostosis in mice, but the sites that are

SUBMITTER: Bok S 

PROVIDER: S-EPMC10799660 | biostudies-literature | 2023 Sep

REPOSITORIES: biostudies-literature